A case of oculo-facio-cardio-dental syndrome with integrated orthodontic-prosthodontic treatment

A case of oculo-facio-cardio-dental syndrome with integrated orthodontic-prosthodontic treatment
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DOI:
10.1597/02-133
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发表时间:
2004-01-01
期刊:
CLEFT PALATE-CRANIOFACIAL JOURNAL
影响因子:
--
通讯作者:
Ohyama, K
Ohyama, K
中科院分区:
其他
文献类型:
--
作者:
Kawamoto, T;Motohashi, N;Ohyama, K

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目的:眼-面-心-牙(OFCD)综合征是一种非常罕见的疾病,需要全面的牙科治疗,因为相关的多个牙齿异常,如犬根肥大,牙列延迟,少牙,恒牙,小牙和巨牙。本报告提出了一个日本女孩(13岁1个月)的OFCD的情况下。我们分析了cephalograms,全景X线片,和牙科模型,并讨论我们的综合牙科修复treatment.Design:牙冠和牙根的大小以及侧头片与日本对照组进行了比较。结果:牙根扩大不仅发生在上、下尖牙,而且发生在上中切牙和下第一前磨牙。巨齿症见于上中切牙、上犬齿、下犬齿和下第一前磨牙。上颌侧切牙存在小齿畸形。头颅侧位测量分析显示,在矢状面和垂直方向上,中面部发育不良,颅底长度显著减少,下颌角增加。从13岁到23岁的正畸治疗过程中的系列侧头X线片显示只有轻微的上颌骨生长和显着的下颌骨向下生长,顺时针旋转,除了明显的唇倾斜的上中切牙。结论:特点牙齿异常连同一个独特的颅面畸形被澄清。成功的口腔修复取得了综合治疗。
Objective: Oculo-facio-cardio-dental (OFCD) syndrome is a very rare condition that requires comprehensive dental management because of associated multiple dental anomalies such as canine radiculomegaly, delayed dentition, oligodontia, persistent primary teeth, microdontia, and macrodontia. This report presents a case of OFCD in a Japanese girl (13 years 1 month old). We analyzed cephalograms, panoramic roentgenograms, and dental casts and discuss our integrated orthodontic-prosthodontic treatment.Design: The sizes of the tooth crown and root as well as lateral cephalograms were compared with those from a Japanese control group. The outcome of orthodontic treatment was evaluated by comparing cephalograms taken before and after treatment.Results: Radiculomegaly was found not only in the upper and lower canines but also the upper central incisors and lower first premolars. Macrodontia was found in the upper central incisors, upper canines, lower canines, and lower first premolars. Microdontia was found in the upper lateral incisor. Lateral cephalometric analysis showed a remarkable hypoplastic midface in both the sagittal and vertical dimensions, coupled with a significantly decreased cranial base length and an increased gonial angle. Serial lateral cephalograms during orthodontic treatment from 13 to 23 years of age demonstrated only slight maxillary growth and significant downward mandibular growth with clockwise rotation in addition to pronounced labial tipping of the upper central incisors.Conclusions: Characteristic dental anomalies together with a unique craniofacial dysmorphology were clarified. Successful oral rehabilitation was achieved by integrated orthodontic-prosthodontic treatment.